Expressive morphosyntax in boys with Fragile X syndrome with and without autism spectrum disorder
Expressive morphosyntax in boys with Fragile X syndrome with and without autism spectrum disorder
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DOI：
10.3109/13682822.2010.487885
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发表时间：
2011-03-01
影响因子：
2.4
通讯作者：
Price, Johanna
中科院分区：
文献类型：
--
作者：
Estigarribia, Bruno;Roberts, Joanne Erwick;Price, Johanna
背景资料：脆性X综合征（FXS）是智力残疾最常见的遗传原因，也是与自闭症相关的最常见的单基因疾病。这种疾病中的语言障碍已有充分的记录，但句法障碍的性质和程度仍不清楚。目的：比较患有FXS且伴有和不伴有自闭症谱系障碍的男孩在动词（VM）和名词（NM）形态句法测量方面的表现与具有相似非语言心理年龄的典型发育男孩的表现。方法和程序：对话样本来自33名患有FXS伴自闭症谱系障碍（FXS-ASD）的男孩，35名患有FXS且无ASD的男孩（FXS-O）和46名典型发育男孩（TD）。在这两个子域中，分别评估了动词和名词形态句法的产生。一个分层线性模型比较了在调整非语言认知，发音技巧，和照顾者的教育后，所有群体的形态句法分数。该模型还测试了组和形态句法subdomain.Outcomes和结果之间的相互作用：男孩与FXS在两组得分低于TD男孩在这两个措施。FXS-O组和FXS-ASD组在两项综合指标上均无差异。所有的协变量均显着相关的morphosyntagnetic scores.Conclusions及影响：部分的形态句法功能障碍FXS可能是由于认知，环境和语音因素。然而，很明显，FXS男孩的表现水平低于预期，这是因为这些语言外因素的差异，无论是在动词还是名词领域。因此，临床干预措施应寻求解决特定的句法目标。
Background: Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability, and the most common single gene disorder associated with autism. Language impairments in this disorder are well documented, but the nature and extent of syntactic impairments are still unclear.Aims: To compare the performance of boys with FXS with and without autism spectrum disorder on measures of verb (VM) and noun (NM) morphosyntax with that of typically developing boys of similar non-verbal mental ages.Methods & Procedures: Conversational samples were obtained from 33 boys with FXS with autism spectrum disorder (FXS-ASD), 35 boys with FXS and no ASD (FXS-O), and 46 typically developing boys (TD). Production of verbal and nominal morphosyntax was assessed separately in these two subdomains. A hierarchical linear model compared morphosyntactic scores in all groups after adjusting for non-verbal cognition, articulatory skill, and caregiver education. The model also tested interactions between group and morphosyntactic subdomain.Outcomes & Results: Boys with FXS in both groups scored lower than the TD boys on both measures. The FXS-O and the FXS-ASD groups did not differ on either composite measure. All covariates were significantly related to morphosyntactic scores.Conclusions & Implications: Part of the morphosyntactic impairment in FXS may be attributable to cognitive, environmental, and speech factors. However, it is clear that boys with FXS perform at levels lower than expected from differences in these extra-linguistic factors alone, across both the verb and the noun domains. Clinical interventions should therefore seek to address specific syntactic targets.